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Encephalopathy Due to Visceral Larva Migrans J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.30.6.580 on 1 December 1967. Downloaded from J. Neurol. Neurosurg. Psychiat., 1967, 30, 580 Encephalopathy due to visceral larva migrans DAVID SUMNER and ELLIS G. F. TINSLEY From the Departments ofNeurology and Pathology, St. James's Hospital, Leeds For some time a syndrome of massive eosino- in America (Milburn and Ernst, 1953), while philia associated with respiratory and other symp- Moore (1962) described a further example which toms has been recognized. The condition carries on presented with convulsions and encephalitis. the whole a good prognosis, is self-limiting and is For an adult to be affected is equally rare and most commonly seen in children, often occurring in Lorentz (1962), who made the diagnosis in a West several members of the same family. Frequently Indian immigrant in London, could find but three observed in the tropics or the subtropics, it has been examples in the literature, and none in an English- variously described as Loeffler's syndrome, tropical born individual. In 1964, Brain and Allan described eosinophilia, familial eosinophilia, or benign eosino- the case of a young Englishwoman who had visceral philic leukaemia. In 1940 this syndrome was shown larva migrans presenting essentially the picture of to be associated with multiple eosinophilic granu- varying focal epilepsy. Although the larva was not guest. Protected by copyright. lomata in the liver, and in 1950, Mercer and his found it seemed likely that the infecting parasite colleagues (Mercer, Lund, Bloomfield, and Caldwell, was a Toxocara and it is of interest that five years 1950) found that with this eosinophilia, Nematode before the onset of her illness she had gone to live larvae were sometimes to be seen in the tissues. In in Calcutta. 1952, Beaver, Snyder, Carrera, Dent, and Lafferty The present example is reported not only because suggested that the syndrome resulted from infection of the paucity of accounts of clinical involvement in man by a Nematode normally parasitic upon a of the nervous system but also because of the lower animal; under these conditions the normal apparent rarity of the condition in the adult. Nematode life cycle does not take place but is blocked by an eosinophilic granulomatous reaction CASE REPORT at the stage where the larvae are in the tissues. At this point systemic symptoms and signs may appear, The patient was a 57-year-old school mistress, teaching in addition to an eosinophilia, and it was for this sixth form mathematics. She had never visited the stage that they coined the term 'visceral larva tropics and has lived all her life in the north of England. migrans'. She had never kept any pets and denied any but the most The condition has been reported most frequently casual contact with cats or dogs, but she was a patho- from the more southern states of the U.S.A. and in logical hoarder and as a result of this had lived for some to clinical features years in considerable squalor. The hoarding had been addition the eosinophilia, the going on for many years and she collected not only new include malaise, pyrexia, bronchitis, 'pneumonitis', and unwanted objects (as, for example, four unused enlargement of the liver (and less commonly the paraffin heaters, a 20-ft. extending ladder, and 87 pound http://jnnp.bmj.com/ spleen), lymphadenopathy, skin rashes, and sub- jars of Bovril), but all rubbish and uneaten food. Nothing cutaneous granulomata. In view of the wide distri- had been placed in a dustbin for 10 years but the full bution of the larvae in the viscera, it would be extent of the hoarding only came to light when she was surprising if the brain were to escape and indeed, admitted to hospital and her house could be visited. Dent, Nichols, Beaver, Carrera, and Staggers in Figure 1, which shows some of the 3,600 full milk 1956, describing the necropsy findings in a child who bottles which were stored in the bedrooms and Figure 2, suffered from this condition but had died from which shows her kitchen, give some idea of what the house was like. Although it is not our contention that another illness, found five larvae per gram of wet her recent encephalopathy was directly related to this on October 2, 2021 by brain tissue. Despite this, clinical involvement of the longstanding psychological abnormality (in the sense of nervous system has rarely been reported. Reviewing sharing the same pathology), it seems reasonable to 58 cases Lewis, Yadav, and Kern (1962) found conclude that the state of her house may well have mention of convulsions in but three patients, one provided a route for Nematode infection. For many in Britain (Dickson and Woodcock, 1959) and two years she had lived apart from her husband, but he 580 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.30.6.580 on 1 December 1967. Downloaded from Encephalopathy due to visceral larva migrans 581 I' i. FIG. 1. The patient's bedroom. FIG. 2. The patient's kitchen. nevertheless visited her about twice a week and was thus although she knew the day and the date, she had no able to say that in September 1965, she began to look ill recollection of the events of the preceding week. She had and at the beginning of October, she stopped work and no knowledge of current affairs and was totally unable retired to bed, where she lay confused and incontinent. to carry out the serial seven test. On the other hand, she The day before her admission to hospital (25 October) knew that the area ofa circle was wrr. On general examina- guest. Protected by copyright. she was found to have a temperature of 103°F. but within tion the only abnormality was in the chest, where there 24 hours she became apyrexial and remained so. She was diminished air entry at the right lung base and in herself could give no clear account of her condition and this area scattered expiratory rhonchi could be heard. rCI#S,#w ; Big -ftc, AC lb ( e I7I0I .e Temporal montage 1 second I 50 pV http://jnnp.bmj.com/ on October 2, 2021 by FIG. 3. The E.E.G. recorded on admission showing an increase in slow wave activity in anterior leads, particularly on overbreathing. J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.30.6.580 on 1 December 1967. Downloaded from 582 David Sumner and Ellis G. F. Tinsley -V:;<Wt>s, Neither the liver nor the spleen was palpable. In the ,1k.11 ~.114- central nervous system the only abnormality (apart of course from confusion) was the presence of blateral A.~~~~~extensor plantar responses. The cerebrospinal fluid was normal, but the E.E.G. showed some diffuse abnormality, particularly on over ~~~~~~~~~~ breathing (Fig. 3). z4>>t^$>t The striking finding on investigation was a considerable JN v {. @ ;"" W g> eosinophilia of 47% of a total W.B.C. count of 9,800 per c.mm., the eosinophilia persisting throughout her $*s--<>sWBa* R g ¢ % stay in hospital, and a search for helminth parasites was A~i....EX.....commenced.m Faeces were examined on many occasions by concentration methods but no ova, larvae, or adult ts;i worms were discovered. Twice the stomach was washed vAI:.t t.ifl ^' '%'>t*v-sbf4out-X. in the early morning and the aspirate similarly : searched;t<;<1!,with negative results. However, an x-ray S %,* Xs e9sskmitx§examination: ;tab of the chest showed a high right diaphragm, 46fi,:whichR suggested that there might be some enlargement t t a :> > * of the liver, although this could never be felt. Liver +"' .-^t)t,.;^t function tests at this time were abnormal, suggesting 4Xt~a~. considerable liver damage: serum bilirubin 0-5 mg./100 44 A g ml.; serum alkaline phosphatase 28 K.A. units; thymol W flo~~~cculation weakly positive; thymol turbidity and zinc sulphate turbidity at the upper limit of normal; S.G.P.T. 127 units; S.G.O.T. 62 units; total serum proteins guest. Protected by copyright. a ~.e ; 6 . ss¢58.ei^e..........6-2f:rt.g/100*R9.W.444ml.;'(albumin 3-3, globulin 2-9); electrophoretic Z.a..I;! A4'tsi7'$.. pattern normal. weeks after admission to hospital a needle biopsy of the liver was performed. A small white nodule was seen in the middle of the specimen, and microsco- FIG. 4. Liver biopsy showing the granuloma and Nematode pically it consisted of an inflammatory granuloma some larva. :e.....*R4¢..,ti...............Three3 mmn. in diameter (Fig. 4). Necrotic liver cells and Temporal montage http://jnnp.bmj.com/ 1.s 10&nVJ 1 second on October 2, 2021 by FIG. 5. The E.E.G. recorded three weeks after admission showing considerable resolution in the changes seen earlier. J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.30.6.580 on 1 December 1967. Downloaded from Encephalopathy due to visceral larva migrans 583 breaking down polymorphs and eosinophils made up volvement in visceral larva migrans, any attempt the centre, together with numerous Charcot-Leyden to delineate a clinical pattern is obviously impossible. crystals. Embedded in the necrotic mass was a Nematode It is of interest, however, that the few previous larva. Surrounding the centre was a narrow band of in eosinophil leucocytes and outside this a broader area of examples the literature presented with epilepsy young fibrous tissue, heavily infiltrated with eosinophils or 'convulsions'. In this connexion, the important and other chronic inflammatory cells. The rest of the work of Woodruff, Bisseru, and Bowe (1966) must liver biopsy showed normal liver tissue.
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